The Scleroderma Patient-Centered Intervention Network Self-Management Program: Protocol for a Randomized Feasibility Trial.

BACKGROUND Systemic sclerosis (SSc), or scleroderma, is a rare disease that often results in significant disruptions to activities of daily living and can negatively affect physical and psychological well-being. Because there is no known cure, SSc treatment focuses on reducing symptoms and disability and improving health-related quality of life (HRQoL). Self-management programs are known to increase self-efficacy for disease management in many chronic diseases. The Scleroderma Patient-centered Intervention Network (SPIN) developed a Web-based self-management program (SPIN self-management; SPIN-SELF) to increase self-efficacy for disease management and to improve HRQoL for patients with SSc. OBJECTIVE The proposed study aims to assess the feasibility of conducting a full-scale randomized controlled trial (RCT) of the SPIN-SELF program by evaluating the trial implementation processes, required resources and management, scientific aspects, and participant acceptability and usage of the SPIN-SELF program. METHODS The SPIN-SELF feasibility trial will be conducted via the SPIN Cohort. The SPIN Cohort was developed as a framework for embedded pragmatic trials using the cohort multiple RCT design. In total, 40 English-speaking SPIN Cohort participants with low disease management self-efficacy (Self-Efficacy for Managing Chronic Disease Scale score ≤7), who have indicated interest in using a Web-based self-management program, will be randomized with a 3:2 ratio into the SPIN-SELF program or usual care for 3 months. Feasibility outcomes include trial implementation processes, required resources and management, scientific aspects, and patient acceptability and usage of the SPIN-SELF program. RESULTS Enrollment of the 40 participants occurred between July 5, 2019, and July 27, 2019. By November 25, 2019, data collection of trial outcomes was completed. Data analysis is underway, and results are expected to be published in 2020. CONCLUSIONS The SPIN-SELF program is a self-help tool that may improve disease-management self-efficacy and improve HRQoL in patients with SSc. The SPIN-SELF feasibility trial will ensure that trial methodology is robust, feasible, and consistent with trial participant expectations. The results will guide adjustments that need to be implemented before undertaking a full-scale RCT of the SPIN-SELF program. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID) DERR1-10.2196/16799.

[1]  B. Thombs,et al.  Challenges and strategies for coping with scleroderma: implications for a scleroderma-specific self-management program , 2019, Disability and rehabilitation.

[2]  J. Poole,et al.  Randomized Controlled Trial to Evaluate an Internet‐Based Self‐Management Program in Systemic Sclerosis , 2019, Arthritis care & research.

[3]  S. Langan,et al.  Protocol for the development of a CONSORT extension for RCTs using cohorts and routinely collected health data , 2018, Research Integrity and Peer Review.

[4]  B. Thombs,et al.  Performance of the Patient-Reported Outcomes Measurement Information System-29 in scleroderma: a Scleroderma Patient-centered Intervention Network Cohort Study , 2017, Rheumatology.

[5]  M. Campbell,et al.  CONSORT 2010 statement: extension to randomised pilot and feasibility trials , 2016, British Medical Journal.

[6]  S. Hopewell,et al.  CONSORT 2010 statement: extension to randomised pilot and feasibility trials. , 2016, Pilot and feasibility studies.

[7]  M. Suarez‐Almazor,et al.  Validation of the Self‐Efficacy for Managing Chronic Disease Scale: A Scleroderma Patient‐Centered Intervention Network Cohort Study , 2016, Arthritis care & research.

[8]  B. Levis,et al.  Psychosocial Aspects of Scleroderma. , 2015, Rheumatic diseases clinics of North America.

[9]  Kate Lorig,et al.  The English and Spanish Self-Efficacy to Manage Chronic Disease Scale measures were validated using multiple studies. , 2014, Journal of clinical epidemiology.

[10]  Sarah J. Shoemaker,et al.  Development of the Patient Education Materials Assessment Tool (PEMAT): a new measure of understandability and actionability for print and audiovisual patient information. , 2014, Patient education and counseling.

[11]  J. Poole,et al.  Taking Charge of Systemic Sclerosis: A Pilot Study to Assess the Effectiveness of an Internet Self‐Management Program , 2014, Arthritis care & research.

[12]  Oliver Distler,et al.  2013 classification criteria for systemic sclerosis: an American College of Rheumatology/European League against Rheumatism collaborative initiative. , 2013, Arthritis and rheumatism.

[13]  R. Steele,et al.  The Scleroderma Patient-centered Intervention Network (SPIN) Cohort: protocol for a cohort multiple randomised controlled trial (cmRCT) design to support trials of psychosocial and rehabilitation interventions in a rare disease context , 2013, BMJ Open.

[14]  J. Poole,et al.  Evaluation of a mail-delivered, print-format, self-management program for persons with systemic sclerosis , 2013, Clinical Rheumatology.

[15]  Julius Sim,et al.  The size of a pilot study for a clinical trial should be calculated in relation to considerations of precision and efficiency. , 2012, Journal of clinical epidemiology.

[16]  R. Chang,et al.  Validity of two new patient‐reported outcome measures in systemic sclerosis: Patient‐reported outcomes measurement information system 29‐item health profile and functional assessment of chronic illness therapy–dyspnea short form , 2011, Arthritis care & research.

[17]  B. Thombs,et al.  Frequency and impact of symptoms experienced by patients with systemic sclerosis: results from a Canadian National Survey. , 2011, Rheumatology.

[18]  B. Thombs,et al.  Psychological health and well‐being in systemic sclerosis: State of the science and consensus research agenda , 2010, Arthritis care & research.

[19]  Jon Nicholl,et al.  Rethinking pragmatic randomised controlled trials: introducing the “cohort multiple randomised controlled trial” design , 2010, BMJ : British Medical Journal.

[20]  R. Landewé,et al.  EULAR recommendations for the treatment of systemic sclerosis: a report from the EULAR Scleroderma Trials and Research group (EUSTAR) , 2009, Annals of the rheumatic diseases.

[21]  D. Moher,et al.  Improving the reporting of pragmatic trials: an extension of the CONSORT statement , 2008, BMJ : British Medical Journal.

[22]  K. Lorig,et al.  The internet-based arthritis self-management program: a one-year randomized trial for patients with arthritis or fibromyalgia. , 2008, Arthritis and rheumatism.

[23]  Arrigo Schieppati,et al.  Why rare diseases are an important medical and social issue , 2008, The Lancet.

[24]  K. Lorig,et al.  Internet-Based Chronic Disease Self-Management: A Randomized Trial , 2006, Medical care.

[25]  H. Kraemer,et al.  Caution regarding the use of pilot studies to guide power calculations for study proposals. , 2006, Archives of general psychiatry.

[26]  Steven A. Julious,et al.  Sample size of 12 per group rule of thumb for a pilot study , 2005 .

[27]  John P. Allegrante,et al.  A review and synthesis of research evidence for self-efficacy-enhancing interventions for reducing chronic disability: implications for health education practice (part II). , 2005, Health promotion practice.

[28]  K. Lorig,et al.  A Review and Synthesis of Research Evidence for Self-Efficacy-Enhancing Interventions for Reducing Chronic Disability: Implications for Health Education Practice (Part I) , 2005, Health promotion practice.

[29]  S Gary Firestein,et al.  Kelley's Textbook of Rheumatology , 2004 .

[30]  K. Lorig,et al.  Self-management education: History, definition, outcomes, and mechanisms , 2003, Annals of behavioral medicine : a publication of the Society of Behavioral Medicine.

[31]  C. Wright,et al.  Self-management approaches for people with chronic conditions: a review. , 2002, Patient education and counseling.

[32]  K. Lorig,et al.  Effect of a self-management program on patients with chronic disease. , 2001, Effective clinical practice : ECP.

[33]  W. Graham,et al.  The importance of conducting and reporting pilot studies: the example of the Scottish Births Survey. , 2001, Journal of advanced nursing.

[34]  T. Al-Sayed,et al.  Clinical features of systemic sclerosis. , 2001, Saudi medical journal.

[35]  N. Black.  CONSORT , 1996, The Lancet.

[36]  A. Bandura.  Self-efficacy: toward a unifying theory of behavioral change. , 1977, Psychological review.

[37]  B. Thombs,et al.  Performance of the Patient-Reported Outcomes Measurement Information System-29 in scleroderma: a Scleroderma Patient-centered Intervention Network Cohort Study. , 2019, Rheumatology.

[38]  A. Ismaila,et al.  A tutorial on pilot studies: the what, why and how , 2010, BMC medical research methodology.

[39]  D. Torgerson,et al.  Designing randomised trials in health, education and the social sciences : an introduction , 2008 .

[40]  A. Schouffoer,et al.  The scleroderma patient-centered intervention network (SPIN): meeting the challenges of research into a rare disease , 2022 .